Behavior in Prader-Willi syndrome: relationship to genetic subtypes and age / Elisabeth M. DYKENS in Journal of Child Psychology and Psychiatry, 49-9 (September 2008)  

Public ISBD [article]  inJournal of Child Psychology and Psychiatry > 49-9 (September 2008) . - p.1001-1008 Titre : Behavior in Prader-Willi syndrome: relationship to genetic subtypes and age Type de document : Texte imprimé et/ou numérique Auteurs : Elisabeth M. DYKENS, Auteur ; Elizabeth ROOF, Auteur Année de publication : 2008 Article en page(s) : p.1001-1008 Langues : Anglais (eng) Mots-clés : Prader-Willi-syndrome genetic-subtypes age CYFIP1 Index. décimale : PER Périodiques Résumé : Background: Some behavioral features of Prader-Willi syndrome (PWS) are associated with the major genetic subtypes of this disorder. While most agree that those with maternal uniparental disomy (UPD) have a distinctive cognitive and psychiatric profile, findings are more controversial regarding possible differences among persons who vary in paternal deletion size. Methods: Caregivers of 88 persons with PWS aged 5 to 51 years (M = 22 years) were administered measures of problem behavior, compulsivity, hyperphagia, and adaptive skills. The sample was well characterized as having relatively large, Type I (n = 26) or smaller, Type II (n = 29) deletions, or UPD (n = 33). Results: No significant behavioral differences were found between the Type I versus Type II deletion groups. Within each genetic subtype, however, differences emerged in how advancing age related to behavior. Although age did not emerge as a significant correlate of behavior in the Type II or UPD groups, in the Type I group age was consistently associated with lower problem behaviors, adaptive skills, and externalizing symptoms. Conclusion: Although differences between deletion subtypes were not found, significant within-subtype differences emerged in relationships between age and behavior. Negative associations between age and behavior in the Type I group only may relate to non-imprinted genes that are deleted in Type I but not Type II cases, including CYFIP1. Altered expression of CYFIP1 is seen in other developmental disabilities, including 15q disorders, and haploinsufficiency of CYFIP1 in Type I PWS cases may be associated with age-related phenotypic effects. Findings underscore the importance of a life-span perspective in phenotypic research. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2008.01913.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=559 [article] Behavior in Prader-Willi syndrome: relationship to genetic subtypes and age [Texte imprimé et/ou numérique] / Elisabeth M. DYKENS, Auteur ; Elizabeth ROOF, Auteur . - 2008 . - p.1001-1008. Langues : Anglais (eng) in Journal of Child Psychology and Psychiatry > 49-9 (September 2008) . - p.1001-1008 Mots-clés : Prader-Willi-syndrome genetic-subtypes age CYFIP1 Index. décimale : PER Périodiques Résumé : Background: Some behavioral features of Prader-Willi syndrome (PWS) are associated with the major genetic subtypes of this disorder. While most agree that those with maternal uniparental disomy (UPD) have a distinctive cognitive and psychiatric profile, findings are more controversial regarding possible differences among persons who vary in paternal deletion size. Methods: Caregivers of 88 persons with PWS aged 5 to 51 years (M = 22 years) were administered measures of problem behavior, compulsivity, hyperphagia, and adaptive skills. The sample was well characterized as having relatively large, Type I (n = 26) or smaller, Type II (n = 29) deletions, or UPD (n = 33). Results: No significant behavioral differences were found between the Type I versus Type II deletion groups. Within each genetic subtype, however, differences emerged in how advancing age related to behavior. Although age did not emerge as a significant correlate of behavior in the Type II or UPD groups, in the Type I group age was consistently associated with lower problem behaviors, adaptive skills, and externalizing symptoms. Conclusion: Although differences between deletion subtypes were not found, significant within-subtype differences emerged in relationships between age and behavior. Negative associations between age and behavior in the Type I group only may relate to non-imprinted genes that are deleted in Type I but not Type II cases, including CYFIP1. Altered expression of CYFIP1 is seen in other developmental disabilities, including 15q disorders, and haploinsufficiency of CYFIP1 in Type I PWS cases may be associated with age-related phenotypic effects. Findings underscore the importance of a life-span perspective in phenotypic research. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2008.01913.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=559

Cognitive and adaptive advantages of growth hormone treatment in children with Prader-Willi syndrome / Elisabeth M. DYKENS in Journal of Child Psychology and Psychiatry, 58-1 (January 2017)  

Public ISBD [article]  inJournal of Child Psychology and Psychiatry > 58-1 (January 2017) . - p.64-74 Titre : Cognitive and adaptive advantages of growth hormone treatment in children with Prader-Willi syndrome Type de document : Texte imprimé et/ou numérique Auteurs : Elisabeth M. DYKENS, Auteur ; Elizabeth ROOF, Auteur ; Hailee HUNT-HAWKINS, Auteur Article en page(s) : p.64-74 Langues : Anglais (eng) Mots-clés : Prader-Willi syndrome  growth hormone treatment  cognition  adaptive behavior Index. décimale : PER Périodiques Résumé : Background People with Prader-Willi syndrome (PWS) typically have mild to moderate intellectual deficits, compulsivity, hyperphagia, obesity, and growth hormone deficiencies. Growth hormone treatment (GHT) in PWS has well-established salutatory effects on linear growth and body composition, yet cognitive benefits of GHT, seen in other patient groups, have not been well studied in PWS. Methods Study 1 included 96 children and youth with PWS aged 4–21 years who naturalistically varied in their exposures to GHT. Controlling for socioeconomic status, analyses compared cognitive and adaptive behavior test scores across age-matched treatment naïve versus growth hormone treated children. Study II assessed if age of treatment initiation or treatment duration was associated with subsequent cognition or adaptive behavior in 127, 4- to 21-year olds with PWS. Study III longitudinally examined cognitive and adaptive behavior in 168 participants who were either consistently on versus off GHT for up to 4–5 years. Results Compared to the treatment naïve group, children receiving GHT had significantly higher Verbal and Composite IQs, and adaptive communication and daily living skills. Children who began treatment before 12 months of age had higher Nonverbal and Composite IQs than children who began treatment between 1 and 5 years of age. Longitudinally, the groups differed in their intercepts, but not slopes, with each group showing stable IQ and adaptive behavior scores over time. Conclusions Cognitive and adaptive advantages should be considered an ancillary benefit and additional justification for GHT in people with PWS. Future efforts need to target apparent socioeconomic inequities in accessing GHT in the PWS population. En ligne : http://dx.doi.org/10.1111/jcpp.12601 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=298 [article] Cognitive and adaptive advantages of growth hormone treatment in children with Prader-Willi syndrome [Texte imprimé et/ou numérique] / Elisabeth M. DYKENS, Auteur ; Elizabeth ROOF, Auteur ; Hailee HUNT-HAWKINS, Auteur . - p.64-74. Langues : Anglais (eng) in Journal of Child Psychology and Psychiatry > 58-1 (January 2017) . - p.64-74 Mots-clés : Prader-Willi syndrome  growth hormone treatment  cognition  adaptive behavior Index. décimale : PER Périodiques Résumé : Background People with Prader-Willi syndrome (PWS) typically have mild to moderate intellectual deficits, compulsivity, hyperphagia, obesity, and growth hormone deficiencies. Growth hormone treatment (GHT) in PWS has well-established salutatory effects on linear growth and body composition, yet cognitive benefits of GHT, seen in other patient groups, have not been well studied in PWS. Methods Study 1 included 96 children and youth with PWS aged 4–21 years who naturalistically varied in their exposures to GHT. Controlling for socioeconomic status, analyses compared cognitive and adaptive behavior test scores across age-matched treatment naïve versus growth hormone treated children. Study II assessed if age of treatment initiation or treatment duration was associated with subsequent cognition or adaptive behavior in 127, 4- to 21-year olds with PWS. Study III longitudinally examined cognitive and adaptive behavior in 168 participants who were either consistently on versus off GHT for up to 4–5 years. Results Compared to the treatment naïve group, children receiving GHT had significantly higher Verbal and Composite IQs, and adaptive communication and daily living skills. Children who began treatment before 12 months of age had higher Nonverbal and Composite IQs than children who began treatment between 1 and 5 years of age. Longitudinally, the groups differed in their intercepts, but not slopes, with each group showing stable IQ and adaptive behavior scores over time. Conclusions Cognitive and adaptive advantages should be considered an ancillary benefit and additional justification for GHT in people with PWS. Future efforts need to target apparent socioeconomic inequities in accessing GHT in the PWS population. En ligne : http://dx.doi.org/10.1111/jcpp.12601 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=298

Intellectual Disabilities and Autism Spectrum Disorder: A Cautionary Note / Elisabeth M. DYKENS

Public ISBD in Autism Spectrum Disorders / David G. AMARAL Titre : Intellectual Disabilities and Autism Spectrum Disorder: A Cautionary Note Type de document : Texte imprimé et/ou numérique Auteurs : Elisabeth M. DYKENS, Auteur ; Miriam LENSE, Auteur Année de publication : 2011 Importance : p.263-269 Langues : Anglais (eng) Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=139 in Autism Spectrum Disorders / David G. AMARAL Intellectual Disabilities and Autism Spectrum Disorder: A Cautionary Note [Texte imprimé et/ou numérique] / Elisabeth M. DYKENS, Auteur ; Miriam LENSE, Auteur . - 2011 . - p.263-269. Langues : Anglais (eng) Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=139

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Intellectual disabilities and child psychiatry: looking to the future / Robert M. HODAPP in Journal of Child Psychology and Psychiatry, 50-1-2 (January/February 2009)  

Public ISBD [article]  inJournal of Child Psychology and Psychiatry > 50-1-2 (January/February 2009) . - p.99-107 Titre : Intellectual disabilities and child psychiatry: looking to the future Type de document : Texte imprimé et/ou numérique Auteurs : Robert M. HODAPP, Auteur ; Elisabeth M. DYKENS, Auteur Année de publication : 2009 Article en page(s) : p.99-107 Langues : Anglais (eng) Mots-clés : Behavioral-phenotype gene–brain-behavior-relations GE-correlation demographics gender aging Index. décimale : PER Périodiques Résumé : We begin this article by examining the role of intellectual disabilities within child psychiatry, highlighting the relatively steady role of disabilities and the recent movement to examine behavior in specific genetic syndromes. We next propose five questions for future work. Questions relate to (1) specifying the nature of gene–brain–behavior connections; (2) delineating environmental effects and gene–environment interactions; (3) understanding behaviors, physical characteristics, health issues, and other personal characteristics by which children with intellectual disabilities influence others; (4) clarifying the roles of gender and aging in behavioral functioning; and (5) working to improve pharmacological, educational, and other interventions and supports for children and their families. Although great advances have occurred over the past 50 years, intellectual disability research lags behind other areas of child psychiatry; much remains to be discovered that might help these children. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2008.02038.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=694 [article] Intellectual disabilities and child psychiatry: looking to the future [Texte imprimé et/ou numérique] / Robert M. HODAPP, Auteur ; Elisabeth M. DYKENS, Auteur . - 2009 . - p.99-107. Langues : Anglais (eng) in Journal of Child Psychology and Psychiatry > 50-1-2 (January/February 2009) . - p.99-107 Mots-clés : Behavioral-phenotype gene–brain-behavior-relations GE-correlation demographics gender aging Index. décimale : PER Périodiques Résumé : We begin this article by examining the role of intellectual disabilities within child psychiatry, highlighting the relatively steady role of disabilities and the recent movement to examine behavior in specific genetic syndromes. We next propose five questions for future work. Questions relate to (1) specifying the nature of gene–brain–behavior connections; (2) delineating environmental effects and gene–environment interactions; (3) understanding behaviors, physical characteristics, health issues, and other personal characteristics by which children with intellectual disabilities influence others; (4) clarifying the roles of gender and aging in behavioral functioning; and (5) working to improve pharmacological, educational, and other interventions and supports for children and their families. Although great advances have occurred over the past 50 years, intellectual disability research lags behind other areas of child psychiatry; much remains to be discovered that might help these children. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2008.02038.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=694

Leisure Activities in Prader-Wlli Syndrome: Implications for Health, Cognition and Adaptive Functioning / Elisabeth M. DYKENS in Journal of Autism and Developmental Disorders, 44-2 (February 2014)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 44-2 (February 2014) . - p.294-302 Titre : Leisure Activities in Prader-Wlli Syndrome: Implications for Health, Cognition and Adaptive Functioning Type de document : Texte imprimé et/ou numérique Auteurs : Elisabeth M. DYKENS, Auteur Article en page(s) : p.294-302 Langues : Anglais (eng) Mots-clés : Prader-Willi syndrome  Adaptive behavior  Recreation  Obesity  Cognition Index. décimale : PER Périodiques Résumé : Although hyperphagia and compulsivity in Prader-Willi syndrome (PWS) are well described, recreation and adaptive skills are relatively unexplored. Parents of 123 participants with PWS (4–48 years) completed measures of their child’s adaptive, recreation, and problem behaviors. Offspring received cognitive testing. Watching TV was the most frequent recreational activity, and was associated with compulsivity and skin picking. BMIs were negatively correlated with physical play, and highest in those who watched TV and played computer games. Computer games and physical activities were associated with higher IQ and adaptive scores. People with PWS and other disabilities need to watch less TV and be more engaged in physical activities, games, and leisure pursuits that are fun, and may bring cognitive or adaptive advantages. En ligne : http://dx.doi.org/10.1007/s10803-012-1462-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=223 [article] Leisure Activities in Prader-Wlli Syndrome: Implications for Health, Cognition and Adaptive Functioning [Texte imprimé et/ou numérique] / Elisabeth M. DYKENS, Auteur . - p.294-302. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 44-2 (February 2014) . - p.294-302 Mots-clés : Prader-Willi syndrome  Adaptive behavior  Recreation  Obesity  Cognition Index. décimale : PER Périodiques Résumé : Although hyperphagia and compulsivity in Prader-Willi syndrome (PWS) are well described, recreation and adaptive skills are relatively unexplored. Parents of 123 participants with PWS (4–48 years) completed measures of their child’s adaptive, recreation, and problem behaviors. Offspring received cognitive testing. Watching TV was the most frequent recreational activity, and was associated with compulsivity and skin picking. BMIs were negatively correlated with physical play, and highest in those who watched TV and played computer games. Computer games and physical activities were associated with higher IQ and adaptive scores. People with PWS and other disabilities need to watch less TV and be more engaged in physical activities, games, and leisure pursuits that are fun, and may bring cognitive or adaptive advantages. En ligne : http://dx.doi.org/10.1007/s10803-012-1462-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=223

Life Satisfaction Among Mothers of Individuals with Prader-Willi Syndrome / Carolyn M. SHIVERS in Journal of Autism and Developmental Disorders, 46-6 (June 2016)  

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A Longitudinal RCT of P-ESDM With and Without Parental Mindfulness Based Stress Reduction: Impact on Child Outcomes / Amy S. WEITLAUF in Journal of Autism and Developmental Disorders, 52-12 (December 2022)  

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TPH2 G/T polymorphism is associated with hyperphagia, IQ, and internalizing problems in Prader–Willi syndrome / Elisabeth M. DYKENS in Journal of Child Psychology and Psychiatry, 52-5 (May 2011)  

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Trajectories of Diurnal Cortisol in Mothers of Children with Autism and Other Developmental Disabilities: Relations to Health and Mental Health / Elisabeth M. DYKENS in Journal of Autism and Developmental Disorders, 43-10 (October 2013)  

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